Parkinson's Disease-Associated Dysarthria: Prevalence, Impact And

Research and Reviews in Parkinsonism Dovepress

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Open Access Full Text Article REVIEW Parkinson’s disease-associated dysarthria: prevalence, impact and management strategies

This article was published in the following Dove Press journal: Research and Reviews in Parkinsonism

Gemma Moya-Galé1 Abstract: Dysarthria is a motor speech disorder of neurological origin and is characterized Erika S Levy2 by deficits in the execution of movement for speech. Close to 90% of individuals with Parkinson’s disease (PD) present with hypokinetic dysarthria, as evidenced by reduced vocal 1Department of Communication Sciences and Disorders, Long Island University, loudness, monotone, reduced fundamental frequency range, consonant and vowel impreci- Brooklyn, NY, USA; 2Department of sion, breathiness and irregular pauses. The presence of these speech deficits negatively Biobehavioral Sciences, Teachers College, impacts intelligibility, functional communication and, ultimately, social participation. The Columbia University, New York, NY, USA aims of this review are to 1) describe the nature of this motor speech disorder and its impact on the ability to communicate effectively, 2) provide an overview of medical approaches to dysarthria management and 3) review research on behavioral treatment techniques aimed at improving the intelligibility and quality of life of individuals with dysarthria secondary to PD. The delivery of speech treatment through telepractice is also examined, as this is a modality particularly well-suited to individuals with the mobility difficulties characteristic For personal use only. of PD. Finally, dysarthria management across languages is considered, representing a relevant new and under-researched area in motor speech disorders. Keywords: Parkinson’s disease, motor speech disorders, dysarthria, speech therapy

Video abstract Introduction Parkinson’s disease (PD) is the second most common neurodegenerative disease following Alzheimer’s disease1 and affects over six million people worldwide.2 In the United States, approximately one million individuals are estimated to suffer from this disease, considered the 14th leading cause of death in the country.3 Although PD is usually developed between the ages of 55 and 65, 4

Research and Reviews in Parkinsonism downloaded from https://www.dovepress.com/ by 24.199.112.9 on 31-May-2019 with an average disease duration of 10 years, the onset of the disease may start before the age of 40.5 With the world population growing and longevity increasing, the need for treatment research on motor speech disorders is also on the rise.6 Point your SmartPhone at the code above. If you have a The aims of this review are to 1) describe the nature of hypokinetic dysarthria, QR code reader the video abstract will appear. Or use: https://youtu.be/AX620v98FnI the motor speech disorder associated with PD and its impact on the ability to communicate effectively, 2) provide an overview of medical approaches to dysarthria management and 3) review research on behavioral treatment techniques aimed at improving the intelligibility and quality of life of individuals with

Correspondence: Gemma Moya-Galé dysarthria secondary to PD (henceforth, individuals with PD). Crosslinguistic Department of Communication Sciences considerations in dysarthria treatment research are discussed. This overview is and Disorders, Long Island University, 1 University Plaza, Brooklyn, NY 11201, not intended to be an exhaustive review of the literature, but rather to report on USA ﬁndings in treatment research in PD and to consider their implications for clinical Tel +1 718 780 4125 Email [email protected] practice.

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Hypokinetic dysarthria due to PD the linguistic domain, the presence of hypomimia, Hypokinetic dysarthria results from dysfunction in the a reduction in facial expression, also limits these indivi- basal ganglia motor loop,7 which causes deficits in the duals’ communicative interactions, social participation regulation of initiation, amplitude and velocity of move- and the quality of their relationships with their care 18 ment. This type of dysarthria is characterized by reduced partners. vocal loudness, monotone, reduced fundamental frequency range, consonant and vowel imprecision, breathiness, short Pharmacological management of rushes of speech and irregular pauses.8,9 Close to 90% of dysarthria due to PD individuals with PD experience voice disorders, while Pharmaceutical and surgical management are approaches 45% experience articulation problems and 20% experience frequently called upon for treatment of motor symptoms fluency disorders.10,11 Although voice disorders may be of PD. However, these have shown less success in the primary concern regarding speech production in the alleviating symptoms of dysarthria than has behavioral early stages of the disease, disfluency and articulation management. Levodopa, the precursor of the neurotrans- impairments also emerge as the neurodegeneration pro- mitter dopamine, is the first medicine typically used in gresses, with motor control deficits affecting articulation the clinical management of PD. The use of levodopa exerting the greatest negative impact on communication in and dopamine agonists to treat motor symptoms such as the final stage of the disease.10 tremors in PD is strongly supported by scientificevi- Physiologically, hypokinetic dysarthria is associated with dence across all stages of the disease.19 However, phar- poor breath support and reduced range of motion of the macological management of dysarthria is less speech articulators,12,13 contributing to perceived undershoot straightforward.20 Early pharmacokinetic studies on the of target sounds.14 These acoustic-perceptual and physiolo- effects of levodopa medication on speech function gical characteristics, therefore, have a detrimental effect on revealed trends toward improvement during the ON ’ phase of medication.21 Ho, Bradshaw and Iansek studied

For personal use only. the individuals intelligibility and, hence, their ability to communicate effectively. Of note, however, in an examina- the effects of levodopa medication on the speech of nine tion of the perspective of individuals with PD on changes in individuals with idiopathic PD and observed an increase their communication, Miller, Noble, Jones and Burn found in sound pressure level (SPL, the acoustic correlate of that even before intelligibility issues arise, the individuals vocal loudness) in the ON phase,22 which was consistent experience negative consequences of early changes in voice with some previously reported findings,23 but contrasted quality and decreased control of their speech output.15 While with an absence of positive response to levodopa to they are usually able to modify their speech for short periods ameliorate hypophonia found in other studies,24,25 sug- of time, their concerns are less about the changes themselves gesting heterogeneity of individual profiles influencing (eg, decreased control of pitch) than about the impact these outcomes. Ho et al also observed an increase in speak- changes have on their self-image and their communication, ing rate as a result of levodopa medication, consistent and their embarrassment when their speech output is not with the upscaling of gain noted for limb movements.22

Research and Reviews in Parkinsonism downloaded from https://www.dovepress.com/ by 24.199.112.9 on 31-May-2019 what they intended. Moreover, the frustration and effort Fundamental frequency and articulation, however, needed to overcome their communicative limitations can remained unchanged. result in social withdrawal. The authors conclude that early A reduced effect on brain activation patterns involved referral of newly diagnosed individuals with PD should be in the production of speech has also been found following the norm, rather than referrals being delayed until frank levodopa medication.26 A recent study of levodopa effects deficits in intelligibility become evident. on speech and voice in 24 individuals in the late stage of Although the motor speech disorder of dysarthria is the disease (ie, with a Schwab and England score of <50 or a primary communication complaint of individuals with a Hoehn and Yahr stage >3 while on medication) found no PD, it is worth noting that other aspects of communica- improvements in speech in response to this medication.12 tion are also affected. For example, difficulties with lex- Thus, current evidence suggests limited and variable suc- ical retrieval and comprehension of figurative language cess in response to pharmacological treatment of dysar- have been reported,16 as well as with grammaticality, thria, leading to clinical reliance primarily on behavioral syntactic complexity and information content.17 Beyond management strategies.

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Surgical management of dysarthria multiple sclerosis (MS) and 12 individuals with PD.41 Like pharmacological management, surgical management Findings from the PD group indicated that when in PD has generally not yielded positive outcomes for instructed to use speech that was twice as loud as their dysarthria. Deep brain stimulation involves electrode usual speaking voice, half of the individuals improved implantation surgery in specific areas of the basal consonantal distinctiveness for stop consonants. ganglia.27 This surgical procedure has been reported as Listeners’ ratings of intelligibility also increased in the an effective treatment option for motor symptoms such loud condition, suggesting a beneficial effect of increas- as dyskinesias in PD in several randomized controlled ing loudness on perceived intelligibility, consistent with trials (RCT);28–30 however, its adverse effects on speech the intelligibility benefits documented for amplified 42 have been consistently reported in the literature.31–36 Other speech. surgical procedures have also been associated with A slower-than-normal speaking rate has been hypothe- a deterioration of speech. For example, in thalamotomies, sized to increase the precision of consonantal articulation, a lesion is made in the thalamus with the goal of improv- phoneme duration and vowel working space, as well as to ing tremors. Thalamotomies have been reported to produce reduce lexical boundary errors, contributing to the reduction hypophonia, reduce speaking rate and lead to word of phonemic and lexical ambiguity experienced by 43 blocking.37,38 This procedure is even thought to have listeners. Nonetheless, cueing for slow speech appears to resulted in some cases of palilalia.39 Consequently, thala- be a less effective strategy for increasing intelligibility in motomy has been abandoned as a treatment option for dysarthria than is cueing for loud speech, likely because PD.20 Similarly, pallidotomy, in which a heated electrical slowing speech rate limits dynamic formant frequency 44 probe is inserted in the globus pallidus, destroying a small changes, negatively impacting speech naturalness and, region of cells in order to alleviate dyskinesias, has not therefore, intelligibility. Not surprisingly, Tjaden and been found to be conducive to improvement in dysarthric Wilding reported greater intelligibility ratings for speech in 20 their loud condition than in their slow or habitual condition.41

For personal use only. speech. In fact, it has been associated with the development of verbal fluency deficits, swallowing difficulties and Clear speech, in contrast, is characterized by exaggerated 45 facial weakness.40 articulation, with concomitant prosodic changes such as increases in vocal intensity and reduction in speaking rate.46 Behavioral management of Cueing for clear speech may involve instructing individuals to speak clearly or to use speech that is twice as clear as their dysarthria usual speaking voice. Tjaden, Sussman and Wilding com- In part because of the poor outcomes for speech of phar- pared the effects of cueing for clear, loud or slow speech on maceutical and surgical management of PD symptoms, scaled intelligibility in 30 individuals with MS and 16 indi- dysarthria management has come to rely on behavioral viduals with PD.45 Only the clear and loud speaking styles approaches. Fortunately, behavioral approaches have were found to improve intelligibility (in both groups of resulted in more positive outcomes, including clinically individuals), suggesting promise for these two techniques meaningful changes revealed in RCTs. Research and Reviews in Parkinsonism downloaded from https://www.dovepress.com/ by 24.199.112.9 on 31-May-2019 for enhancing communication in individuals with dysarthria.

Speech cueing studies Speech treatment studies Two types of clinically relevant research, speech cueing Unlike speech cueing studies, speech treatment studies studies and speech treatment studies, aim to shed light examine changes from performance at baseline to perfor- on effective behavioral approaches to dysarthria manage- mance following treatment. During testing, speakers are ment. We discuss speech cueing studies ﬁrst, in which not provided with any speech cues. Thus, any gains in investigators provide speech cues such as “loud” or performance represent learned behaviors rather than “slow” or “clear” and compare effects of these cues on immediate responses to cues. Traditional behavioral treat- acoustics and/or intelligibility of dysarthric speech. For ment for dysarthria addresses all speech subsystems: example, Tjaden and Wilding found support, in particu- respiratory drive, phonation, articulation, prosody and lar, for cueing for loud speech in their examination of resonance.47 Immediately following such subsystem treat- the effects of speech cues on the acoustics and intellig- ments, positive results have been found with the more ibility of utterances produced by 15 individuals with intensive treatment protocols.48

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Speech treatments with a specific speech subsystem tar- a group of 45 individuals with PD (33 males and get, rather than targeting multiple subsystems, have also been 12 females).58 While LSVT LOUD focuses on the single developed for hypokinetic dysarthria, with varying degrees target of voice to address the respiratory and phonatory of success. These primarily include focus on increased vocal subsystem deficits that are characteristic of dysarthric loudness or articulatory working space,49,50 although other speech,49,58–60 the intensive respiratory treatment was speech subsystems have also been explored, as described in designed to maximize inspiration and expiration and the following section. achieve increased volume of subglottal air pressure. Respiratory treatment has been implemented in the man- Visual feedback on breathing patterns was provided to agement of dysarthria, with variable success. The aim of this participants during some of the tasks. Statistically signifi- therapeutic approach is to increase respiratory support in cant improvements pre-to-post treatment were found in order to produce sufficient subglottal air pressure for both (LSVT LOUD and RES) groups for various mea- speech.47 Posture control is one of the key characteristics of sures, such as conversational SPL and perceptual self- behavioral respiratory treatment for dysarthria. Positioning ratings of monotonicity. However, overall, LSVT LOUD individuals in supine or prone positions is thought to increase yielded a more consistent and greater increase in funda- their subglottal air pressure (thus increasing their vocal mental frequency variation and vocal intensity, and indi- intensity).51 Despite the importance of adequate respiratory viduals who received LSVT LOUD also reported support for speech, respiratory treatments alone have not a reduced impact of PD on their communication. yielded statistically significant improvement in vocal func- Several physiological benefits have been associated with tion in dysarthria.52 LSVT LOUD, such as vocal quality and articulation,61,62 Another treatment target that has been examined is increased fundamental frequency range (ie, prosodic inflec- resonance. For example, Wenke, Theodoros and tions) and enhanced resonance.49 Recently, an RCT with Cornwell studied ten individuals with velopharyngeal 64 individuals with PD compared SPL across three groups: incompetence (VPI) and nonprogressive dysarthria sec- LSVT LOUD, LSVT ARTIC (an intensive treatment proto- For personal use only. ondary to PD, who were randomly assigned to either col targeting articulation through increased movement ampli- a traditional (TRAD) treatment group or an intensive tude of the speech articulators) and an untreated subset of voice-focused group.53 While the traditional treatment individuals with PD. Results from this RCT showed signifi- group focused on exaggerated articulation, oromotor exer- cant increases in SPL in the individuals in the LSVT LOUD cises within speech tasks, breathing, resonance and pro- group at 1 and 7 months post-treatment, compared to those in sody, the voice-focused group focused on increasing SPL the LSVT ARTIC and untreated groups.63 Furthermore, and fundamental frequency range with a regimen of high- although the speakers in both treated groups obtained higher intensity exercises. Trends toward decreased hypernasality Modified Communication Effectiveness Index scores at immediately after treatment were found in the voice- 1-month post-treatment, only the individuals in LSVT focused group, suggesting potential for a voice-focused LOUD maintained treatment effects on their overall commu- approach for improving velopharyngeal function in PD. nication ratings at the 7 months follow-up, suggesting

Research and Reviews in Parkinsonism downloaded from https://www.dovepress.com/ by 24.199.112.9 on 31-May-2019 With its respiratory–laryngeal subsystem target of voice, a prolonged benefit for SPL and overall communicative the Lee Silverman Voice Treatment (LSVT LOUD) is the effectiveness of intensive speech treatment targeting voice. only speech treatment with Level I evidence for improving Speech intelligibility, too, has generally shown gains vocal function in PD.54 This program elicits maximum vocal following LSVT LOUD. For example, significant intellig- effort during sustained phonations, maximum frequency ibility increases have been reported post-treatment at the range exercises and functional speech tasks and is designed sentence level in stimuli presented to naïve listeners at equal- to increase subglottal air pressure, improve vocal fold adduc- ized intensity levels and in competing noise.64,65 tion and articulatory movements and enhance vocal tract Additionally, Ramig, Countryman, Thompson and Horii configurations.49,54 LSVT LOUD is based on principles of found significant improvements in pre-to-post ratings of motor learning, primarily repetition, intensity, specificity and overall intelligibility (by individuals with PD and their saliency, believed to enhance neural plasticity through an families)58 following this voice-targeted treatment (but see acquired habit of motor routines.55–57 studies by El Sharkawi et al and Ramig et al).66,67 Moreover, Ramig et al compared the voice-focused LSVT LOUD in a recent RCT comparing LSVT LOUD, LSVTARTIC and with an intensive respiratory (RES) treatment in a RCT of an untreated group, ease of understanding ratings by 117

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listeners increased with both experimental treatments, but not this method was effective in reducing speech rate and for the untreated group. The more rigorous intelligibility eliminating palilalia.75 Similarly, Downie, Low and measure of transcription accuracy increased significantly Lindsay found positive effects of a portable delayed audi- for LSVT LOUD, but not for LSVT ARTIC, providing tory feedback device on reduction of speaking rate in stronger support for the voice-targeted approach.68 2 individuals with PD (out of 11); however, the learned The intensive schedule of 4 days of treatment for 4 skill did not generalize without the use of the device; thus, weeks required by the LSVT LOUD protocol may not be the individuals needed to rely on its continued use to feasible in many treatment settings. Thus, treatment studies maintain the benefits.76 with adaptations to the schedule have also been conducted in English and in other languages, yielding positive results. Dysarthria management beyond the LSVT-X involves treatment twice a week for eight conse- individual: group therapy approaches cutive weeks, following the hierarchy and tasks established Group treatment is also often implemented in university 69 fi in LSVT LOUD. Signi cant increases in SPL have been clinics and beyond, with the aim of improving speech com- fi found following LSVT-X, as well as signi cant improve- munication in PD in a larger, supportive setting. De Angelis ments in judgments of vocal quality, speaking rate, intona- et al investigated the effects of 13 group therapy sessions on tion, naturalness and articulatory clarity in individuals with voice variables in 20 individuals with PD.50 Treatment PD. LSVT-X was implemented in a study of the effects of focused on increasing phonatory function through the imple- pharmaceutical and speech treatment on prosody in 10 mentation of a high-effort program based on pushing exer- – 70 Brazilian-Portuguese speaking individuals with PD. The cises during phonation, as well as overarticulation techniques results were optimal for participants who were tested while to maximize articulatory precision. Positive changes follow- taking levodopa. After sixteen 50-min sessions twice ing speech treatment were reported, indicating greater laryn- a week, the participants revealed increased fundamental geal efficiency. Specifically, the study reported an increase in frequency and intensity and reduced utterance duration, For personal use only. maximum phonation times and vocal intensity, a decrease in fi suggesting that a modi ed schedule of intensive vocal s/z ratio measures and a reduction in self-perceived deviant fi exercises may also lead to acoustic bene ts. vocal characteristics (eg, monotone or strained-strangled For similar scheduling reasons, as well as for ease of voice quality). An improvement in speech intelligibility access, adaptations to the delivery modality of LSVT LOUD was also found as measured by participants’ self- have also been implemented, with positive results found with evaluations, suggesting promise for such group treatment. the use of teletherapy techniques. Teletherapy approaches using LSVT LOUD range from the use of videophones and videoconferencing through Skype to more sophisticated tech- Crosslinguistic research on speech niques that allow for the precise measurement of SPL, funda- treatment for PD – mental frequency and duration.71 73 Findings from Most of the studies described above, as well as the vast major- a randomized controlled noninferiority online trial showed ity found in the literature, report on outcomes of American

Research and Reviews in Parkinsonism downloaded from https://www.dovepress.com/ by 24.199.112.9 on 31-May-2019 comparable results between the traditional face-to-face LSVT English-speaking individuals with PD. Despite the estimated LOUD protocol and its online version, providing clinical prevalence of over six million individuals with PD worldwide, validity of the online modality for speech rehabilitation in little research has been conducted on speech treatment for PD individuals with PD.74 in languages other than English. In a study of Spanish speakers with PD, Moya-Galé et al investigated the effects of LSVT LOUD on conversational intelligibility and self-perceptions of Augmentative and alternative daily communicative capabilities.77 Subjective and objective communication intelligibility measures (including the rigorous intelligibility Beyond treatment retraining speech production in adults measure of listeners’ orthographic transcription accuracy) with PD, the use of augmentative and alternative devices revealed substantial and signiﬁcant gains in both variables may be implemented to enhance overall communication.47 post-treatment. These ﬁndings, therefore, support the imple- Helm investigated the effects of using a pacing board for 2 mentation of this intensive treatment approach in Spanish- weeks on the speaking rate of a single individual with speaking populations and raise the question of whether vocal postencephalitic Parkinson syndrome and concluded that loudness increases conversational intelligibility universally.

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Acoustic studies of the effects of voice-targeted treat- Disclosure ment in languages other than English have also been con- The authors report no conflicts of interest in this work. ducted. Whitehill, Kwan, Lee and Chow investigated the effects of LSVT LOUD in 12 Cantonese-speaking individuals with PD.78 Results revealed significant improve- References ments in both vocal loudness and intonation. However, 1. Dorsey ER, Constantinescu R, Thompson JP, et al. Projected number of people with Parkinson disease in the most populous nations, 2005 lexical tone was relatively intact, as measured by tone through 2030. Neurology. 2007;68(5):384–386. doi:10.1212/01. acoustics (ie, fundamental frequency configurations) and wnl.0000247740.47667.03 perceptual analysis (ie, listeners’ transcription of isolated 2. Ray Dorsey E, Elbaz A, Nichols E, et al. Global, regional, and national burden of Parkinson’s disease, 1990–2016: a systematic syllables and identification of error tones in phrases). This analysis for the Global Burden of Disease Study 2016. Lancet outcome is in contrast to the well-documented deficits Neurol. 2018;17:939–953. doi:10.1016/S1474-4422(18)30295-3 3. Xu J, Murphy SL, Kochanek KD, Bastian B, Arias E. Deaths: final experienced by individuals with PD at the laryngeal data for 2016. Nvss. 2018;67:1–75. level.79,80 Significant acoustic changes at the segmental 4. Rizek P, Kumar N, Jog MS. An update on the diagnosis and treatment – level pre-to-post treatment (eg, increased vowel duration of Parkinson disease. Cmaj. 2016;188(16):1157 1165. doi:10.1503/ cmaj.151179 and increased vowel space area) have been also reported in 5. Quinn N, Critchley P, Marsden CD. Young onset Parkinson’s disease. other languages (eg, Quebecois French) following an Mov Disord. 1987;2(2):73–91. doi:10.1002/mds.870020201 81 fi 6. Duffy JR. Motor speech disorders: where will we be in 10 years? intensive voice-based treatment. These ndings are in Semin Speech Lang. 2016;37(3):219–224. doi:10.1055/s-0036- agreement with previously reported results in the English 1584154 literature.82 The similarities in intelligibility gains as 7. Albin RL, Young AB, Penney JB. The functional anatomy of basal ganglia disorders. Trends Neurosci. 1989;12:366–375. a result of increased vocal effort in English and Spanish, 8. Darley FL, Aronson AE, Brown JR. Clusters of deviant speech as well as the observed increases in vowel space in English dimensions in the dysarthrias. J Speech Hear Res. 1969;12 (3):462–496. and Quebecois French, post-treatment, are preliminary 9. Duffy JR. Motor Speech Disorders: Substrates, Differential evidence of potentially language-universal effects of treat- Diagnosis, and Management. 3rd ed. St. Louis: Mosby; 2013. For personal use only. ment, warranting further investigation. Language-specific 10. Ho AK, Iansek R, Marigliani C, Bradshaw JL, Gates S. Speech impairment in a large sample of patients with Parkinson’s disease. constraints of treatment still remain largely unexplored, Behav Neurol. 1998;11(3):131–137. but differences in articulation and prosody across lan- 11. Logemann JA, Fisher HB, Boshes B, Blonsky ER. Frequency and cooccurrence of vocal tract dysfunctions in the speech of a large sample guages are likely to render differential effects of treatment of Parkinson patients. JSpeechHearDisord. 1978;43(1):47–57. in PD.83 We maintain that it is of utmost clinical impor- 12. Fabbri M, Guimarães I, Cardoso R, et al. Speech and voice response ’ tance for further research to address questions of whether to a levodopa challenge in late-stage Parkinson s disease. Front Neurol. 2017;8:432. doi:10.3389/fneur.2017.00432 any speech treatment benefits in individuals with PD might 13. Yorkston KM, Beukelman DR, Strand EA, Hakel M. Management of be 1) universal, regardless of their language background or Motor Speech Disorders in Children and Adults. 3rd ed. Austin: Pro- fi Ed; 2010. 2) constrained by language-speci c characteristics. 14. McAuliffe MJ, Ward EC, Murdoch BE. Speech production in Parkinson’s disease: I. An electropalatographic investigation of ton- gue-palate contact patterns. Clin Linguist Phon. 2006;20(1):1–18. Conclusion and future directions doi:10.1080/02699200400001044

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